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Background: The COVID-19 pandemic negatively affected child and adolescent mental health and at the end of the pandemic (April 2022) child mental health had not returned to pre-pandemic levels. We investigated whether this observed increase in mental health problems has continued, halted, or reversed after the end of the pandemic in children from the general population and in children in psychiatric care., Methods: We collected parent-reported and child-reported data at two additional post-pandemic time points (November/December 2022 and March/April 2023) in children (8-18 years) from two general population samples ( N  = 818-1056 per measurement) and one clinical sample receiving psychiatric care ( N  = 320-370) and compared these with data from before the pandemic. We collected parent-reported data on internalizing and externalizing problems with the Brief Problem Monitor and self-reported data on Anxiety, Depressive symptoms, Sleep-related impairments, Anger, Global health, and Peer relations with the Patient-Reported Outcomes Measurement Information System (PROMIS ® )., Results: In the general population, parents reported no changes in externalizing problems but did report higher internalizing problems post-pandemic than pre-pandemic ( p  < 0.001). Children also reported increased mental health problems post-pandemic, especially in anxiety and depression, to a lesser extent in sleep-related impairment and global health, and least in anger (all p s < 0.01). In the clinical sample, parents reported higher internalizing ( p  < 0.001), but not externalizing problems post-pandemic compared to the start of the pandemic. Children reported greatest increases in problems in anxiety, depression, and global health, to a lesser extent on sleep-related impairment, and least on anger (all p s < 0.05)., Conclusions: Child mental health problems in the general population are substantially higher post-pandemic compared to pre-pandemic measurements. In children in psychiatric care mental health problems have increased during the pandemic and are substantially higher post-pandemic than at the start of the pandemic. Longitudinal and comparative studies are needed to assess what the most important drivers of these changes are., Competing Interests: MB is supported by a European Research Council consolidator Grant (WELL‐BEING 771057 PI Bartels). The authors have declared that they have no competing or potential conflicts of interest. All authors have seen and approved the manuscript., (© 2023 The Authors. JCPP Advances published by John Wiley & Sons Ltd on behalf of Association for Child and Adolescent Mental Health.)

Background: Steroid-sensitive nephrotic syndrome (SSNS) is associated with a relapsing-remitting course that can be stressful for parents. As little is known of parental distress at the first onset of SSNS, this study aims to describe parental distress and everyday problems in mothers and fathers of a child with newly diagnosed SSNS participating in a randomized controlled trial of levamisole added to corticosteroids., Methods: To assess distress, the Distress Thermometer for Parents (DT-P) was used, which includes questions on distress (thermometer score 0-10, ≥ 4 "clinical distress") and presence of everyday problems in six domains: practical, social, emotional, physical, cognitive, and parenting. The DT-P was completed 4 weeks after the onset of SSNS. Total sum and individual items of everyday problems were compared with reference data from mothers and fathers of the Dutch general population., Results: There was no difference in clinically elevated parental distress between SSNS mothers (n = 37) and fathers (n = 25) and reference parents. Compared to reference fathers, fathers of a child with SSNS scored significantly higher on emotional problems (P = 0.030), while mothers experienced more parenting problems (P = 0.002). Regression analyses showed that lower parental age and having a girl with SSNS were significantly associated with more practical problems and higher distress thermometer scores, respectively., Conclusions: Four weeks after onset, SSNS mothers and fathers experience equal distress as reference parents. However, both parents endorsed significantly more everyday problems. Therefore, monitoring parental distress, even in the first weeks of the disease, could contribute to timely interventions and prevent worsening of problems., Clinical Trial Registry: Dutch Trial Register ( https://onderzoekmetmensen.nl/en/trial/27331 ). A higher resolution version of the Graphical abstract is available as Supplementary information., (© 2023. The Author(s).)

This study assessed HRQoL and emotional and behavioral difficulties (EBD) and associated variables in children with first onset SSNS. While relapsing steroid-sensitive nephrotic syndrome (SSNS) in children is associated with lower health-related quality of life (HRQoL), little is known about first onset. Four weeks after onset, children (2-16 years) and/or their parents who participated in a randomized placebo-controlled trial, completed the Pediatric Quality of Life Inventory 4.0 (PedsQL) and Strengths and Difficulties Questionnaire (SDQ) to measure HRQoL and EBD, respectively. Total and subscale scores and the proportion of children with impaired HRQoL (> 1 SD below the mean of the reference group) or SDQ clinical scores (< 10 th and > 90 th percentile) were compared to the Dutch general population (reference group). Regression analyses were used to identify associated variables. Compared to the reference group, children 8-18 years reported significantly lower total HRQoL, and physical and emotional functioning. A large proportion (> 45%) of these children had impaired HRQoL. There were no differences in HRQoL between children 2-7 years and the reference group, except for higher scores on social functioning (5-7 years). Similar proportions of SSNS and reference children scored within the clinical range of SDQ subscales. Age, sex, and steroid side-effects were negatively associated with HRQol and/or EBD.   Conclusion: This study showed that HRQoL and EBD are affected in children of different ages with first onset SSNS. This calls for more awareness from healthcare providers and routinely monitoring of HRQoL and EBD in daily clinical care to prevent worsening of symptoms.   Clinical trial registry: Netherlands Trial Register ( https://trialsearch.who.int/ ; NTR7013), date of registration: 02 June 2018. What is Known: •  Health-related quality of life (HRQoL) is lower and emotional and behavioral difficulties (EBD) is more affected in children with frequently-relapsing and steroid-dependent nephrotic syndrome. What is New: •  HRQoL and EBD are affected in children with first onset steroid-sensitive nephrotic syndrome compared to a reference group of the Dutch general population. • To what extent HRQoL and EBD are affected depends on the age of the patient., (© 2023. The Author(s).)

The aim of the study was to assess internalizing problems before and during the pandemic with data from Dutch consortium Child and adolescent mental health and wellbeing in times of the COVID-19 pandemic, consisting of two Dutch general population samples (GS) and two clinical samples (CS) referred to youth/psychiatric care. Measures of internalizing problems were obtained from ongoing data collections pre-pandemic (N GS  = 35,357; N CS  = 4487) and twice during the pandemic, in Apr-May 2020 (N GS  = 3938; clinical: N CS  = 1008) and in Nov-Dec 2020 (N GS  = 1489; N CS  = 1536), in children and adolescents (8-18 years) with parent (Brief Problem Monitor) and/or child reports (Patient-Reported Outcomes Measurement Information System ® ). Results show that, in the general population, internalizing problems were higher during the first peak of the pandemic compared to pre-pandemic based on both child and parent reports. Yet, over the course of the pandemic, on both child and parent reports, similar or lower levels of internalizing problems were observed. Children in the clinical population reported more internalizing symptoms over the course of the pandemic while parents did not report differences in internalizing symptoms from pre-pandemic to the first peak of the pandemic nor over the course of the pandemic. Overall, the findings indicate that children and adolescents of both the general and clinical population were affected negatively by the pandemic in terms of their internalizing problems. Attention is therefore warranted to investigate long-term effects and to monitor if internalizing problems return to pre-pandemic levels or if they remain elevated post-pandemic., (© 2022. The Author(s).)

Background: Classical galactosemia (CG), an inborn error of galactose metabolism, results in long-term complications including cognitive impairment and movement disorders, despite early diagnosis and dietary treatment. Two decades ago, lower motor-, cognitive- and social health related quality of life (HRQoL) was demonstrated in pediatric and adult patients. Since then, the diet has been relaxed, newborn screening was implemented and new international guidelines resulted in major changes in follow-up. The aim of this study was to assess HRQoL of CG by means of online self- and/or proxy-HRQoL-questionnaires focusing on the main areas of concern of CG (i.e. anxiety, depression, cognition, fatigue, social- and upper extremity function) within the patient-reported outcomes measurement information system (PROMIS®) and generic HRQoL-questionnaires (TAPQOL, TACQOL, TAAQOL)., Results: Data of 61 Dutch patients (aged 1-52 years) were collected and compared to available Dutch or US reference populations. On the PROMIS-questionnaires, children reported more fatigue (P = 0.044), lower function in upper extremities (P = 0.021), more cognitive difficulties (P = 0.055, d = 0.56) and higher anxiety (P = 0.063, d = 0.52) than reference children although the latter findings remained non-significant. Parents of CG patients reported lower quality of peer relationships of their children (P < 0.001). Both children and parents reported lower cognitive functioning (P = 0.005, P = 0.010) on the TACQOL. Adults reported on PROMIS domains lower cognitive functioning (P = 0.030), higher anxiety (P = 0.004) and more fatigue (P = 0.026). Cognitive difficulties were reported on the TAAQOL by adults (P < 0.001), as well as physical-, sleeping and social difficulties., Conclusions: CG remains to impact the HRQoL of pediatric and adult patients negatively on several domains including cognition, anxiety, motor function and fatigue. A lower social health was mainly reported by parents, and not by patients themselves. The Covid-19 pandemic might have amplified the results on anxiety although higher levels of anxiety fit pre-pandemic findings. The reported fatigue is a new finding in CG. Because the effect of lockdown fatigue could not be eliminated and fatigue is a frequent finding in patients with chronic disorders, future studies are warranted. Clinicians and researchers should be attentive to both pediatric and adult patients, and the age-dependent difficulties they might encounter., (© 2023. The Author(s).)

Background: The COVID-19 pandemic has had an acute impact on child mental and social health, but long-term effects are still unclear. We examined how child mental health has developed since the start of the COVID-19 pandemic up to 2 years into the pandemic (April 2022)., Methods: We included children (age 8-18) from two general population samples ( N  = 222-1333 per measurement and N  = 2401-13,362 for pre-covid data) and one clinical sample receiving psychiatric care ( N  = 334-748). Behavioral questionnaire data were assessed five times from April 2020 till April 2022 and pre-pandemic data were available for both general population samples. We collected parent-reported data on internalizing and externalizing problems with the Brief Problem Monitor and self-reported data on Anxiety, Depressive symptoms, Sleep-related impairments, Anger, Global health, and Peer relations with the Patient-Reported Outcomes Measurement Information System (PROMIS®)., Results: In all samples, parents reported overall increased internalizing problems, but no increases in externalizing problems, in their children. Children from the general population self-reported increased mental health problems from before to during the pandemic on all six PROMIS domains, with generally worst scores in April 2021, and scores improving toward April 2022 but not to pre-pandemic norms. Children from the clinical sample reported increased mental health problems throughout the pandemic, with generally worst scores in April 2021 or April 2022 and no improvement. We found evidence of minor age effects and no sex effects., Conclusions: Child mental health in the general population has deteriorated during the first phase of the COVID-19 pandemic, has improved since April 2021, but has not yet returned to pre-pandemic levels. Children in psychiatric care show worsening of mental health problems during the pandemic, which has not improved since. Changes in child mental health should be monitored comprehensively to inform health care and policy., Competing Interests: MB is supported by a European Research Council consolidator Grant (WELL‐BEING 771057 PI Bartels). The authors have declared that they have no competing or potential conflicts of interest., (© 2023 The Authors. JCPP Advances published by John Wiley & Sons Ltd on behalf of Association for Child and Adolescent Mental Health.)

Background: In the last decades, pediatric patient engagement has received growing attention and its importance is increasingly acknowledged. Pediatric patient engagement in health care can be defined as the involvement of children and adolescents in the decision-making of daily clinical care, research and intervention development. Although more attention is paid to pediatric patient engagement, a comprehensive overview of the activities that have been done regarding pediatric patient engagement and the changes over time is lacking. Therefore, the aim of this study is to provide an overview of the literature about pediatric patient engagement., Methods: The methodological framework of Arksey & O'Malley was used to conduct this scoping review. The bibliographic databases Medline, Embase, and PsycINFO were searched for eligible articles. All retrieved articles were screened by at least two researchers in two steps. Articles were included if they focused on pediatric patient engagement, were carried out in the context of clinical care in pediatrics, and were published as full text original article in English or Dutch. Data (year of publication, country in which the study was conducted, disease group of the participants, setting of pediatric patient engagement, used methods, and age of participants) were extracted, synthesized, and tabulated., Results: A total of 288 articles out of the 10,714 initial hits met the inclusion criteria. Over the years, there has been an increase in the number of studies that engage pediatric patients. Pediatric patients, especially patients with multiple conditions or oncology patients, were most involved in studies in the United States, United Kingdom, and Canada. Pediatric patients were most often asked to express their views on questions from daily clinical care and the individual interview was the most used method. In general, the extent to which pediatric patients are engaged in health care increases with age., Discussion: This scoping review shows that there is an increasing interest in pediatric patient engagement. However, lack of uniformity about the definition of pediatric patient engagement and clear information for clinicians hinders engagement. This overview can inform clinicians and researchers about the different ways in which pediatric patient engagement can be shaped and can guide them to engage pediatric patients meaningfully in their projects., (© 2023. The Author(s).)

Background: Patient reported outcome measures (PROMs) may be useful for patients with benign liver tumours and cysts (BLTC) to evaluate the impact of treatment and/or guide shared decision making. Yet, a set of PROMs relevant to patients with BLTC is currently unavailable. In this study, we selected a PROMs set for patients with BLTC., Methods: Potentially relevant patient reported outcomes (PROs) were selected by psychologist-researchers based on keywords used or suggested by participants of two virtual focus groups meetings consisting of thirteen female BLTC patients with a median age of 50 years. Subsequently, patients were asked to report their most relevant PROs. PROMs identified by systematic literature review and computerized adaptive tests (CATs) in the Patient-Reported Outcomes Measurement Information System (PROMIS) were considered in selecting the final PROMs set to assess relevant outcomes., Results: The most important PROs were: insecurity/anxiety (11/12 patients), pain (9/12 patients), fatigue (8/12 patients), and limitations in daily life (5/12 patients). The literature review included 23 studies, which used various generic and disease-specific PROMs, often not measuring (all) relevant PROs. The final selected PROMs set included numerical rating scales for pain, two questions on overall health and quality of life and four PROMIS CATs., Conclusions: A PROMs set generically and efficiently measuring outcomes relevant for patients with BLTC was developed and may be used in future research and clinical practice., (© 2022. The Author(s).)

Purpose: The study aimed to compare the psychosocial development of young adult survivors of childhood cancer (YACCS) with a norm group of young adults from the general population., Methods: From 2017 to 2020, 558 YACCS (18-30 years, 51% female, 10.9% CNS cancer) who participated in the Dutch Childhood Cancer Survivor Study (DCCSS) LATER cohort (diagnosed 1963-2001) part 2 completed the Course of Life Questionnaire (CoLQ), assessing the achievement of milestones. Items were grouped into the scales autonomy, psychosexual, and social development. Differences between YACCS and norm group were examined with ANOVA and Cohen's d (CoLQ scales) and with logistic regression analysis and odds ratio (OR) (CoLQ items), for the total group and YACCS of CNS cancer., Results: The total group of YACCS did not report a less favorable psychosocial development than the norm group. YACCS of CNS cancer scored lower than the norm group (p < 0.001) on the scales autonomy (d = - 0.36) and psychosexual (d = - 0.46). Additionally, on half of the items of autonomy (0.25 ≤ OR ≤ 0.34), psychosexual (0.30 ≤ OR ≤ 0.48), and social (0.23 ≤ OR ≤ 0.47) development, YACCS of CNS cancer were less likely (p < 0.01) than the norm group to have achieved the milestones., Conclusion: Overall, psychosocial development of YACCS was as favorable as the norm, but YACCS of CNS cancer were at risk of an unfavorable psychosocial development in all domains. Monitoring psychosocial development should be included in the standards of psychosocial care, especially for CNS cancer patients and survivors, to be able to trace delay. Personalized interventions should be offered to improve the psychosocial development in an early stage., (© 2022. The Author(s).)

The psychosocial consequences of growing up with Heritable Connective Tissue Disorders (HCTD) are largely unknown. We aimed to assess Health-Related Quality of Life (HRQoL) and mental health of children and adolescents with HCTD. This observational multicenter study included 126 children, aged 4-18 years, with Marfan syndrome (MFS, n = 74), Loeys-Dietz syndrome (n = 8), molecular confirmed Ehlers-Danlos syndromes (n = 15), and hypermobile Ehlers-Danlos syndrome (hEDS, n = 29). HRQoL and mental health were assessed through the parent and child-reported Child Health Questionnaires (CHQ-PF50 and CHQ-CF45, respectively) and the parent-reported Strengths and Difficulties Questionnaire. Compared with a representative general population sample, parent-reported HRQoL of the HCTD-group showed significantly decreased Physical sum scores (p < 0.001, d = 0.9) and Psychosocial sum scores (p = 0.024, d = 0.2), indicating decreased HRQoL. Similar findings were obtained for child-reported HRQoL. The parent-reported mental health of the HCTD-group showed significantly increased Total difficulties sum scores (p = 0.01, d = 0.3), indicating decreased mental health. While the male and female MFS- and hEDS-subgroups both reported decreased HRQoL, only the hEDS-subgroup reported decreased mental health. In conclusion, children and adolescents with HCTD report decreased HRQoL and mental health, with most adverse outcomes reported in children with hEDS and least in those with MFS. These findings call for systematic monitoring and tailored interventions., (© 2022 The Authors. American Journal of Medical Genetics Part A published by Wiley Periodicals LLC.)

(1) Background: Health-related quality of life (HRQoL) is lower in patients with chronic kidney disease (CKD) compared to the general population. In 2011, the KLIK PROM portal was implemented in the Emma Children's Hospital to monitor and discuss HRQoL in daily care. This study describes and assesses the implementation and use of the KLIK PROM portal in the pediatric nephrology department. (2) Methods: CKD patients (self-report, if 8-18 years of age) and their parents (proxy-report, if 1-8 years) were invited to complete HRQoL patient-reported outcome measures (PROMs): TNO-AZL Preschool children Quality Of Life (TAPQOL) or Pediatric Quality of Life Inventory for Children (PedsQL). The PROMs were completed before and discussed during outpatient consultations. The adaptation rate-the proportion of patients/parents who were invited and completed at least one PROM-was calculated. Reported HRQoL scores of CKD patients were compared to the general population. (3) Results: In total, 142 patients (proxy- and self-report) were invited, 112 patients completed at least one PROM (adaptation rate 79%). Patients ( n = 84 with informed consent for scientific use) with CKD reported lower HRQoL and HRQoL was more often impaired compared to the general Dutch population. (4) Conclusions: The implementation of KLIK was successful and its use is feasible for daily care. Using KLIK, HRQoL problems can be easily identified and monitored.

Background: Little is known about health-related quality of life (HRQoL) in young children with sickle cell disease living in a European country., Methods: A retrospective cross-sectional evaluation of TNO-AZL Preschool Children Quality of Life questionnaire (TAPQOL, 0-1 year) and Pediatric Quality of Life Inventory (PedsQL, 2-7 years) data was conducted. Study participants included caregivers of children with sickle cell disease aged 0-7 years attending the sickle cell centre at the Erasmus Medical Center or the Amsterdam University Medical Centers between April 2012 and October 2020. Comparisons were made with normative data on HRQoL in the general paediatric population., Results: The study enrolled 136 caregivers of 136 children. In children aged 0-5 years, no significant differences emerged between children with sickle cell disease and the general population. However, in children aged 5-7 years, children with sickle cell disease scored significantly lower on all subscales except for emotional functioning. Multiple regression models showed a negative association between age and HRQoL. No association was found between HRQoL and disease severity or sociodemographic characteristics., Conclusions: This study demonstrates that HRQoL is negatively correlated with age in young children with sickle cell disease with a significantly lower HRQoL in 5- to 7-year-olds when compared to the general population. Our study underlines the importance of measuring HRQoL in young children to identify patients with impaired HRQoL early in life in order to be able to intervene accordingly. Future research should focus on deepening the knowledge of factors influencing HRQoL in children with sickle cell disease., (© 2021 The Authors. Pediatric Blood & Cancer published by Wiley Periodicals LLC.)

Objectives: The aim of the study was to describe the longitudinal development of health-related quality of life (HRQOL) and fatigue in children with chronic intestinal failure (CIF) on home parenteral nutrition (PN) and compare these children to the general population., Methods: Prospective, observational study conducted over 7 years in patients suffering from CIF receiving home PN from 2 tertiary hospitals in the Netherlands. Every 6 months, parents (if child <8 years old) or patients (if child ≥8 years old) completed 2 questionnaires: Pediatric Quality of Life Inventory 4.0 (PedsQL) Generic and Fatigue on the KLIK (kwaliteit van leven in kaart [Dutch Acronym for Quality of Life in Clinical Practice]) Patient Reported Outcome Measures portal, which were compared with the general population. Linear mixed models (LMMs) were constructed to investigate the course of HRQOL over time., Results: Thirty-five patients were included (40% girls). At time of last KLIK contact, patients received HPN for a median of 5.3 years (interquartile range [IQR]: 2.9-9.7). In total, 272 questionnaires were completed. PedsQL generic total score for ages 5 to 7 and 8 to 12 years was significantly lower than the general population (P < 0.01 for both age groups) with effect sizes of 0.73 and 0.71, respectively. PedsQL fatigue total score for ages 5 to 7 years was also significantly lower (P = 0.01; effect size 0.70). LMMs for PedsQL Generic and Fatigue total score 2 to 7 and 8 to 18 years showed no significant coefficient for duration of home PN., Conclusions: Children suffering from CIF receiving home PN ages 5 to 12 years report lower HRQOL scores than the general population. HRQOL and fatigue do not change during long-term treatment with home PN in these children., Competing Interests: S.N. and M.T. have received an investigator-initiated research grant from the Baxter Healthcare Corporation, Deerfield, IL and an ESPGHAN networking grant. M.B. has been a consultant for Shire, Norgine, Tramedico, Allergan, Danone, Novalac, FrieslandCampina, and Sensus, and has received honoraris from Abbott as a speaker. The remaining authors report no conflicts of interest., (Copyright © 2021 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of the European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and the North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition.)

Introduction: The KLIK Patient Reported Outcome Measures (PROM) portal ( www.hetklikt.nu ) has been implemented since 2011 in clinical practice in over 20 Dutch hospitals. Patients and/or parents complete PROMs before the outpatient consultation and answers are subsequently discussed by clinicians during consultation. This study aims to provide insight into patients' and parents' perspective on the use of the KLIK PROM portal in order to optimize its implementation in pediatric clinical practice., Methods: Patients (12-19 years) and parents (of children 0-19 years) from the Emma Children's Hospital were invited to participate. A mixed-method design was used; (1) Focus groups were held and analyzed using thematic analysis in psychology, (2) a questionnaire was sent out and analyzed using descriptive statistics., Results: (1) Eight patients and 17 parents participated. Patients mentioned that KLIK has an attractive layout. However, PROMs were sometimes considered irrelevant and repetitive. Parents valued that KLIK provides insight into their child's functioning, but they were not satisfied with the extent to which PROMs were discussed by clinicians. (2) 31 patients and 130 parents completed the questionnaire. Overall, patients and parents reported a satisfaction score of 7.9/10 and 7.3/10, respectively. 81% of patients and 74% of parents indicated that KLIK is easy to use., Conclusion: Patients and parents are generally satisfied with KLIK, however, points of improvement were mentioned. These are currently being addressed by e.g., upgrading the KLIK website, implementing PROMIS item banks in KLIK to reduce irrelevancy and repetitiveness of PROMs, and implementation strategies to improve the discussion-rate. In this way, implementation of the KLIK PROM portal can be further optimized, with the ultimate goal to improve quality of care., (© 2021. The Author(s).)

Purpose: Patient-reported outcome and experience measures (PROMs/PREMs) are well established in research for many health conditions, but barriers persist for implementing them in routine care. Implementation science (IS) offers a potential way forward, but its application has been limited for PROMs/PREMs., Methods: We compare similarities and differences for widely used IS frameworks and their applicability for implementing PROMs/PREMs through case studies. Three case studies implemented PROMs: (1) pain clinics in Canada; (2) oncology clinics in Australia; and (3) pediatric/adult clinics for chronic conditions in the Netherlands. The fourth case study is planning PREMs implementation in Canadian primary care clinics. We compare case studies on barriers, enablers, implementation strategies, and evaluation., Results: Case studies used IS frameworks to systematize barriers, to develop implementation strategies for clinics, and to evaluate implementation effectiveness. Across case studies, consistent PROM/PREM implementation barriers were technology, uncertainty about how or why to use PROMs/PREMs, and competing demands from established clinical workflows. Enabling factors in clinics were context specific. Implementation support strategies changed during pre-implementation, implementation, and post-implementation stages. Evaluation approaches were inconsistent across case studies, and thus, we present example evaluation metrics specific to PROMs/PREMs., Conclusion: Multilevel IS frameworks are necessary for PROM/PREM implementation given the complexity. In cross-study comparisons, barriers to PROM/PREM implementation were consistent across patient populations and care settings, but enablers were context specific, suggesting the need for tailored implementation strategies based on clinic resources. Theoretically guided studies are needed to clarify how, why, and in what circumstances IS principles lead to successful PROM/PREM integration and sustainability., (© 2020. The Author(s).)

Purpose: The KLIK Patient-Reported Outcome Measure (PROM) portal is an evidence-based intervention implemented in clinical practice in > 25 Dutch hospitals for patients (children and adults) who regularly visit the outpatient clinic. Implementation science frameworks can be used to understand why implementation succeeded or failed, to structure barriers and enablers, and to develop implementation strategies to overcome barriers. This paper aimed to (A) retrospectively describe determinants of successful KLIK PROM implementation using the Consolidated Framework for Implementation Research (CFIR), and (B) identify current barriers and match implementation strategies., Methods: (A) The KLIK implementation process was described retrospectively based on literature and experience, using the 39 CFIR constructs organized in five general domains: intervention characteristics, outer setting, inner setting, characteristics of individuals, and implementation process. (B) The CFIR-Expert Recommendations for Implementing Change (ERIC) Implementation Strategy Matching tool identified current barriers in the KLIK implementation and matched implementation strategies that addressed the identified barriers., Results: (A) The most prominent determinants of successful KLIK PROM implementation lie in the following CFIR domains: intervention characteristics (e.g., easy to use), characteristics of individuals (e.g., motivation), and process of implementation (e.g., support). (B) 13 CFIR constructs were identified as current barriers for implementing the KLIK PROM portal. The highest overall advised ERIC strategy for the specific KLIK barriers was to identify and prepare champions., Conclusion: Using an implementation science framework, e.g., CFIR, is recommended for groups starting to use PROMs in clinical care as it offers a structured approach and provides insight into possible enablers and barriers., (© 2020. The Author(s).)

Purpose: Since 2011, the evidence-based KLIK Patient Reported Outcome Measure (PROM) portal has been implemented in clinical practice in > 20 Dutch hospitals. Patients and/or parents complete PROMs on Health Related Quality of Life, symptoms and psychosocial functioning before their outpatient consultation. Answers are converted into an ePROfile and discussed by clinicians during consultation to monitor well-being over time and detect problems early. This study aims to get insight into the KLIK implementation from the clinician's perspective., Methods: As part of the KLIK implementation process, annual meetings were held with multidisciplinary teams to evaluate the use of KLIK. An online questionnaire was sent regarding (1) overall satisfaction, (2) feeling competent to discuss PROMs, (3) use of KLIK during the consultation, (4) influence of KLIK on the consultation, (5) usability of the KLIK PROM portal, (6) satisfaction with PROMs and feedback, and (7) support of the KLIK expert team. Open questions about (dis)advantages were included. Descriptive analyses were used., Results: One hundred and forty-eight clinicians (response-rate 61%) from 14 hospitals in the Netherlands participated. Results show that: (1) clinicians report an overall satisfaction of median = 69/100 (visual analogue scale), (2) 85.8% feel competent discussing the ePROfile, (3) 70.3% (almost) always discuss the ePROfile, (4) 70.3% think that KLIK improves consultation, (5) 71.6% think KLIK is easy to use, (6) 80.4% are satisfied with the feedback of the overall KLIK ePROfile, (7) 71.6% experience sufficient support of the KLIK team., Conclusion: Participating clinicians are generally satisfied with KLIK. Improvements to the KLIK PROM portal are now realized based on the mentioned disadvantages (e.g., shorten PROM completion by use of PROMIS and integrating KLIK with Electronic Health Records)., (© 2020. The Author(s).)

Purpose: During the COVID-19 pandemic in the Netherlands, governmental regulations resulted in a lockdown for adults as well as children/adolescents. Schools were closed and contact with other people was limited. In this cross-sectional, population-based study, we aimed to investigate the mental/social health of children/adolescents during COVID-19 lockdown., Methods: Two representative samples of Dutch children/adolescents (8-18 years) before COVID-19 (2018, N = 2401) and during lockdown (April 2020, N = 844) were compared on the Patient-Reported Outcomes Measurement Information System (PROMIS) domains: global health, peer relationships, anxiety, depressive symptoms, anger, sleep-related impairment by linear mixed models and calculating relative risks (RR (95% CI)) for the proportion of severe scores. Variables associated with worse mental/social health during COVID-19 were explored through multivariable regression models. The impact of COVID-19 regulations on the daily life of children was qualitatively analyzed., Results: Participants reported worse PROMIS T-scores on all domains during COVID-19 lockdown compared to before (absolute mean difference range 2.1-7.1 (95% CI 1.3-7.9). During lockdown, more children reported severe Anxiety (RR = 1.95 (1.55-2.46) and Sleep-Related Impairment (RR = 1.89 (1.29-2.78) and fewer children reported poor Global Health (RR = 0.36 (0.20-0.65)). Associated factors with worse mental/social health were single-parent family, ≥ three children in the family, negative change in work situation of parents due to COVID-19 regulations, and a relative/friend infected with COVID-19. A large majority (> 90%) reported a negative impact of the COVID-19 regulations on daily life., Conclusion: This study showed that governmental regulations regarding lockdown pose a serious mental/social health threat on children/adolescents that should be brought to the forefront of political decision-making and mental healthcare policy, intervention, and prevention., (© 2021. The Author(s).)

Background: To reduce the burden of completing Patient-Reported Outcome Measures (PROMs), PROMIS® Computerized Adaptive Tests (CATs) are being implemented in pediatric clinical practice. We aimed to develop recommendations for visual feedback options for PROMIS CATs on individual item and domain score level as an evidence-based feedback recommendation for PROMIS CATs is lacking., Methods: Focus groups were held with clinicians who use the KLIK PROM portal. Literature-based feedback options were provided to initiate group discussion. Data was analyzed using thematic coding method. Additionally, a questionnaire was sent out to assess patients' (12-18y) and parents' (child 0-18y) preference for individual item feedback. Data was analyzed using descriptive statistics., Results: Six focus groups were held (N = 28 clinicians). Regarding individual item feedback, showing the complete item bank, with only responses to administered items in traffic light colors was preferred. For domain scores, line graphs were preferred, including numerical (T-)scores, reference and cut-off lines, and traffic light colors. Separate graphs per domain, ranked in order of importance and harmonization of directionality ('higher = better') were considered important. Questionnaire results (N = 31 patients/N = 131 parents) showed that viewing their own item responses was preferred above receiving no item feedback by 58.1% of the patients and 77.1% of the parents., Conclusions: Based on the outcomes and after discussion with the Dutch-Flemish PROMIS National Center, recommendations for PROMIS CAT feedback options were developed. PROMIS CATs can now be used in clinical practice to help clinicians monitor patient outcomes, while reducing the burden of completing PROMs for patients significantly., (© 2021. The Author(s).)

Background: The COVID-19 lockdown increases psychological problems in children and adolescents from the general population. Here we investigate the mental and social health during the COVID-19 lockdown in children and adolescents with pre-existing mental or somatic problems. Methods: We included participants (8-18 years) from a psychiatric ( N = 249) and pediatric ( N = 90) sample, and compared them to a general population sample ( N = 844). Measures were assessed during the first lockdown (April-May 2020) in the Netherlands. Main outcome measures were Patient-Reported Outcomes Measurement Information System (PROMIS®) domains: Global Health, Peer Relationships, Anxiety, Depressive Symptoms, Anger, and Sleep-Related Impairment, as reported by children and youth. Additionally, socio-demographic variables, COVID-19-related questions, changes in atmosphere at home from a parent and child perspective, and children's experiences of lockdown regulations were reported by parents. Results: On all measures except Global Health, the pediatric sample reported least problems. The psychiatric sample reported significantly more problems than the general population sample on all measures except for Anxiety and Peer Relationships. Having a COVID-19 affected friend/relative and a COVID-19 related change in parental work situation negatively moderated outcome, but not in the samples with pre-existing problems. All parents reported significant decreases in atmosphere at home, as did children from the general population. Conclusion: We observed significant differences in mental and social health between three child and adolescent samples during the COVID-19 pandemic lockdown and identified COVID-19-related factors influencing mental and social health., Competing Interests: JB has served as a member of the advisory board of Medice, Roche, Servier, Angelini and has received speaker fee from Takeda and Medice. The remaining authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest., (Copyright © 2021 Zijlmans, Teela, van Ewijk, Klip, van der Mheen, Ruisch, Luijten, van Muilekom, Oostrom, Buitelaar, Hoekstra, Lindauer, Popma, Staal, Vermeiren, van Oers, Haverman and Polderman.)

Aim: To compare Health-Related Quality of Life (HRQOL) of paediatric patients with newly collected HRQOL data of the general Dutch population, explore responses to individual items and investigate variables associated with HRQOL., Methods: Children (8-12y) and adolescents (13-17y) from the general population (N = 966) and from a paediatric population (N = 1209) completed the Pediatric Quality of Life Inventory (PedsQL TM ) online via the KLIK Patient-Reported Outcome Measures portal. PedsQL TM scale scores were compared between groups with independent t tests, by age group and gender. Responses to PedsQL TM items were explored using descriptive analyses. Linear regression analyses were performed to determine which variables were associated with HRQOL., Results: Paediatric patients reported worse HRQOL than the general population on all PedsQL TM scales (p ≤ .001, d = 0.20-1.03), except social functioning, and a high proportion reported problems on PedsQL TM items, for example, 'I have trouble sleeping'. Younger age, female gender and school absence were negatively associated with HRQOL (β = -0.37-0.10, p ≤ .008)., Conclusion: Paediatric patients reported lower HRQOL than the general population, and school absence, female gender and younger age were associated with lower HRQOL. The results underline the importance to structurally monitor paediatric patients' HRQOL in clinical practice to detect problems and offer the right help on time., (©2021 Foundation Acta Paediatrica. Published by John Wiley & Sons Ltd.)

Objectives: Health-related quality of life (HRQOL) of children with avoidant restrictive food intake disorder (ARFID) is impaired., Aim: To measure HRQOL and distress of parents of children with ARFID., Methods: Cross-sectional cohort study. Parents of children with ARFID, visiting our multidisciplinary feeding team, completed questionnaires on the online Quality of Life in Clinical Practice portal; the Questionnaire for Adult's Health Related Quality of Life to assess parental HRQOL and the Distress Thermometer for Parents. Reference groups of parents of healthy (HC) and chronically ill children (CIC) were used., Results: Eighty-five mothers and 62 fathers of 89 children with ARFID (58% female, median age 1.9 years) were included (response rate 68%). No differences were found regarding HRQOL in 11 of 12 domains between parents of children with ARFID and HC. Mothers of children with ARFID reported significantly higher HRQOL regarding pain and fathers a significantly lower HRQOL on depressive emotions compared to HC. No differences were found in overall and clinical distress scores between parents of children with ARFID and HC/CIC. Mothers of children with ARFID had significantly higher distress scores regarding cognitive problems compared to HC and parenting problems in children <2 years compared to HC/CIC. Significantly higher distress scores on parenting problems in children <2 years were found in fathers of children with ARFID compared to HC/CIC., Conclusion: Most HRQOL and distress scores of parents of children with ARFID were comparable to reference groups. Since parents of children with ARFID perceive a lack of understanding and support from the environment, professionals should suggest peer support through patient's organizations. Furthermore, it is important to offer professional support since parents indicated that they would like to talk to a professional about their situation., Competing Interests: The authors report no conflicts of interest, (Copyright © 2021 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of the European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and the North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition.)

Marfan syndrome (MFS) is a multisystemic, autosomal dominant connective tissue disorder that occurs de novo in 25%. In many families, parent and child(ren) are affected, which may increase distress in parents. To assess distress, 42 mothers (29% MFS) and 25 fathers (60% MFS) of 43 affected children, completed the validated screening-questionnaire Distress thermometer for parents of a chronically ill child, including questions on overall distress (score 0-10; ≥4 denoting "clinical distress") and everyday problems (score 0-36). Data were compared to 1,134 control-group-parents of healthy children. Mothers reported significantly less overall distress (2, 1-4 vs. 3, 1-6; p = .049; r = -.07) and total everyday problems (3, 0-6 vs. 4, 1-8; p = .03; r = -.08) compared to control-group-mothers. Mothers without MFS reported significantly less overall distress compared to mothers with MFS, both of a child with MFS (1, 0-4 vs. 3.5, 2-5; p = .039; r = -.17). No significant differences were found between the father-groups, nor between the group of healthy parents of an affected child living together with an affected partner compared to control-group-parents. No differences in percentages of clinical distress were reported between mothers and control-group-mothers (33 vs. 42%); fathers and control-group-fathers (28 vs. 32%); nor between the other groups. Distress was not associated with the children's MFS characteristics. Concluding, parents of a child with MFS did not show more clinical distress compared to parents of healthy children. However, clinical distress was reported in approximately one-third and may increase in case of acute medical complications. We advise monitoring distress in parents of a child with MFS to provide targeted support., (© 2020 The Authors. American Journal of Medical Genetics Part A published by Wiley Periodicals LLC.)

Objective: To assess the psychometric properties of 8 pediatric Patient-Reported Outcomes Measurement Information System (PROMIS) item banks in a clinical sample of children with juvenile idiopathic arthritis (JIA)., Methods: A total of 154 Dutch children (mean ± SD age 14.4 ± 3.0 years; range 8-18 years) with JIA completed 8 pediatric version 1.0 PROMIS item banks (anger, anxiety, depressive symptoms, fatigue, pain interference, peer relationships, physical function mobility, physical function upper extremity) twice and the Pediatric Quality of Life Inventory (PedsQL) and the Childhood Health Assessment Questionnaire (C-HAQ) once. Structural validity of the item banks was assessed by fitting a graded response model (GRM) and inspecting GRM fit (comparative fit index [CFI], Tucker-Lewis index [TLI], and root mean square error of approximation [RMSEA]) and item fit (S-X 2 statistic). Convergent validity (with PedsQL/C-HAQ subdomains) and discriminative validity (active/inactive disease) were assessed. Reliability of the item banks, short forms, and computerized adaptive testing (CAT) was expressed as the SE of theta (SE[θ]). Test-retest reliability was assessed using intraclass correlation coefficients (ICCs) and smallest detectable change., Results: All item banks had sufficient overall GRM fit (CFI >0.95, TLI >0.95, RMSEA <0.08) and no item misfit (all S-X 2 P > 0.001). High correlations (>0.70) were found between most PROMIS T scores and hypothesized PedsQL/C-HAQ (sub)domains. Mobility, pain interference, and upper extremity item banks were able to discriminate between patients with active and inactive disease. Regarding reliability, PROMIS item banks outperformed legacy instruments. Post hoc CAT simulations outperformed short forms. Test-retest reliability was strong (ICC >0.70) for all full-length item banks and short forms, except for the peer relationships item bank., Conclusion: The pediatric PROMIS item banks displayed sufficient psychometric properties for Dutch children with JIA. PROMIS item banks are ready for use in clinical research and practice for children with JIA., (© 2019 The Authors. Arthritis Care & Research published by Wiley Periodicals, Inc. on behalf of American College of Rheumatology.)

Objectives: Parents of children with a chronic illness (CI) are at risk for psychosocial problems. The aim of this study was to refine an existing face-to-face intervention into an online psychosocial group intervention for parents by (1) exploring which themes are important, (2) determine what type of intervention parents would like and (3) assess parents' practical preferences., Methods: Parents of children with a CI (0-18 years) were invited to complete an online questionnaire. To acquire more in-depth information, focus groups and telephone interviews were conducted. Descriptive statistics were used., Results: 272 parents (mean age = 43.1 years, 85% female) participated. Three focus groups (15 parents) and seven telephone interviews were conducted. Most important themes were: the CI of the child, family functioning, taking care of yourself, relationships with others and practical support. Parents preferred a group with parents of children in the same age category. At first, parents preferred face-to-face contact. After an explanation and demonstration of an online intervention, parents became more positive about online support, mostly because they could participate from home., Conclusions for Practice: Parents have a need for psychosocial support focusing on different themes. Professionals should explain and demonstrate an online intervention to parents. Based on these results, Op Koers Online for parents was developed. An RCT to assess feasibility and effectiveness of the intervention is currently running.

Introduction: Idiopathic nephrotic syndrome (INS) is characterised by a high relapse rate up to 80% after initial response to standard therapy with corticosteroids. Steroid toxicity is common and causes a great burden of disease that negatively influences the health-related quality of life (HRQoL). Recently, studies have shown that levamisole, an anthelminthic drug, significantly improves relapse-free survival in children with frequent relapses or steroid dependency. Compared with other steroid-sparing drugs, levamisole has relatively few side effects. We hypothesise that adding levamisole to standard therapy with corticosteroids in children with a first episode of INS will prevent relapses, decrease cumulative dosage of steroids used and improve HRQoL. This paper presents the study protocol for the LEARNS study (LEvamisole as Adjuvant therapy to Reduce relapses of Nephrotic Syndrome)., Methods and Analysis: An international, double-blind, placebo-controlled randomised trial will be conducted in 20 participating hospitals in the Netherlands and Belgium. Participants (n=92) with a first episode of INS, aged 2-16 years, who achieve remission after 4 weeks of oral prednisolone will be randomly assigned (1:1) to receive either levamisole 2.5 mg/kg alternate day or placebo added to prednisolone (18-week tapering schedule) for a total of 24 weeks. Follow-up will be until 2 years after first presentation. Additionally, parents and/or children will fill out five HRQoL questionnaires. Primary outcome of the LEARNS study is occurrence of relapses within 12 months after first presentation. Secondary outcomes include time to first relapse, cumulative steroid dose after 2 years, safety parameters and quality of life scores., Ethics and Dissemination: The trial was approved by the Medical Ethical Committee. Results of the study will be published in a peer-reviewed journal., Trial Registration Number: NL6826, 2017-001025-41., Competing Interests: Competing interests: None declared., (© Author(s) (or their employer(s)) 2019. Re-use permitted under CC BY. Published by BMJ.)

Background & Aims: Parents of children with intestinal failure, dependent on Home Parenteral Nutrition (HPN), may experience psychosocial problems due to the illness and intensive treatment of their child. Literature concerning psychosocial problems is scarce. Therefore, we aimed to investigate Health-Related Quality of Life (HRQOL), levels of anxiety, depression, distress and everyday problems of these mothers and fathers., Methods: A multicenter study was conducted among 37 mothers and 25 fathers of 37 children on HPN (response-rate 37/49 = 76%, mean age children = 5.1 years, SD = 4.6). Parents completed three questionnaires to measure different outcomes on the KLIK website (www.hetklikt.nu): the TNO-AZL QOL Questionnaire (TAAQOL) to measure HRQOL, the Hospital Anxiety and Depression Scale (HADS) to measure anxiety and depression, and the Distress Thermometer for Parents (DT-P) to measure distress. Scores were compared to Dutch reference mothers and fathers using Mann-Whitney U-tests., Results: No differences were found in HRQOL, measured by the TAAQOL, between HPN parents compared to the reference groups, except for the subscale 'depressive emotions' for mothers (p = .01) and 'daily activities' for fathers (p = .04). HPN mothers reported higher levels of depression compared to reference mothers (p = .001). In addition, HPN mothers and fathers reported higher levels of distress than reference mothers (p = .001) and fathers (p = .03). HPN mothers reported significantly more problems in the practical, emotional, cognitive and parenting domains, fathers in the social, emotional and parenting domains., Conclusions: On HRQOL, anxiety and depression, HPN parents generally did not show much differences compared to reference parents. However, when asked about parental distress and everyday problems, HPN treatment of their child seems highly stressful for some parents and influences daily functioning. Therefore, structural screening for parental psychosocial problems in clinical practice, e.g. using the DT-P, is necessary in order to improve the well-being of both these parents and their children dependent on HPN., (Copyright © 2018 Elsevier Ltd and European Society for Clinical Nutrition and Metabolism. All rights reserved.)

Introduction: This paper describes the different ways in which information is being fed back to health care providers (HCPs) using the Dutch evidence-based KLIK Patient-Reported Outcome Measures (PROMs) portal (www.hetklikt.nu). KLIK was initially developed for children with a chronic illness and their parents, and recently expanded for use in adult health care. Patients complete online PROMs at home about their health-related quality of life (HRQOL), symptoms and/or psychosocial functioning before an outpatient consultation with their HCP. PROMs are subsequently converted into an individual KLIK electronic (e)PROfile and discussed during the consultation to facilitate systematic attention for HRQOL., Methods for Pro Score Interpretation: KLIK includes a variety of different PROMs. The KLIK ePROfile initially consisted of literal representations of the individual PROM items in the European traffic light colors (red, orange, green) and only one graph. Over the years, the KLIK ePROfile evolved into a broader spectrum of feedback options; (1) literal representation of individual items, (2) summary scores, and (3) graphic representations (4 options)., Developing Recommendations for Acting on Pro Results: The primary goal of KLIK is to longitudinally monitor patient and parent self-reported or proxy-reported health outcomes, which means that the focus of KLIK is not mainly on recommending actions for PRO results. However, there are several aids that help interpretation of the KLIK ePROfile and recommendations are given for actions based on PROM scores. The main aid is the KLIK training, which includes a theoretical and a practical part. In the training, 2 tools-a decision tree and a summary of information about the KLIK ePROfile-are given to the HCPs to assist them in using KLIK., Discussion: Customization of a PROM portal is needed for each different patient group and clinical setting. Because the KLIK website is flexible, every PROM (with good psychometric qualities and permission of the publisher) can be built in. However, implementing and feeding back PROMs in pediatric health care encompasses different challenges, because different versions of the same PROM are needed for children of different ages, as well as proxy and self-report versions.

Background: Quality of life (QoL) is a prominent outcome measure in mental health. However, conventional methods for QoL assessment rely heavily on language-based communication and therefore may not be optimal for all individuals with severe mental health problems. In addition, QoL assessment is usually based on a fixed number of life domains. This approach conflicts with the notion that QoL is influenced by individual values and preferences. A digital assessment app facilitates both the accessibility and personalization of QoL assessment and may, therefore, help to further advance QoL assessment among individuals with severe mental health problems., Objective: This study focused on the development of an innovative, visual, and personalized QoL assessment app for people with severe mental health problems: the QoL-ME., Methods: This study targeted 3 groups of individuals with severe mental health problems: (1) people with psychiatric problems, (2) people treated in forensic psychiatry, and (3) people who are homeless. A group of 59 participants contributed to the 6 iterations of the cocreative development of the QoL-ME. In the brainstorming stage, consisting of the first iteration, participants' previous experiences with questionnaires and mobile apps were explored. Participants gave their feedback on initial designs and wireframes in the second to fourth iterations that made up the design stage. In the usability stage that comprised the final 2 iterations, the usability of the QoL-ME was evaluated., Results: In the brainstorming stage, participants stressed the importance of privacy and data security and of receiving feedback when answering questionnaires. Participants in the design stage indicated a preference for paging over scrolling, linear navigation, a clean and minimalist layout, the use of touchscreen functionality in various modes of interaction, and the use of visual analog scales. The usability evaluation in the usability stage revealed good to excellent usability., Conclusions: The cocreative development of the QoL-ME resulted in an app that corresponds to the preferences of participants and has strong usability. Further research is needed to evaluate the psychometric quality of the QoL-ME and to investigate its usefulness in practice., (©David C Buitenweg, Ilja L Bongers, Dike van de Mheen, Hans AM van Oers, Chijs van Nieuwenhuizen. Originally published in JMIR Mental Health (http://mental.jmir.org), 28.03.2019.)

Background: Mucopolysaccharidosis type III (MPS III or Sanfilippo syndrome) is a lysosomal storage disease resulting in progressive neurocognitive decline during childhood and early demise. Its diagnosis may have a great impact on parents, potentially leading to psychosocial problems such as anxiety, depression, parental distress, and posttraumatic stress., Methods: Twenty-six mothers and 19 fathers of 34 Dutch MPS III patients completed the "Hospital Anxiety and Depression Scale" (HADS), the "Distress Thermometer for Parents" (DT-P), and the "Self-Rating Scale for Posttraumatic Stress Disorders" (SRS-PTSD). Independent-sample T-tests and chi-square tests were used to assess differences between parents of MPS III patients and reference groups regarding anxiety and depression (HADS), distress (DT-P), and posttraumatic stress (SRS-PTSD)., Results: Mothers met the criteria for clinically relevant anxiety (50%) and depression (34.6%) more frequently compared to reference mothers (p = 0.001). Fathers more often met the criteria for clinically relevant depression (36.8%) compared to reference fathers (p = 0.022). Clinically relevant distress was highly prevalent in mothers (84.6%) and fathers (68.4%) of MPS III patients compared to reference parents (p < 0.01). Finally, the prevalence of PTSD was strikingly higher in both mothers (26.9%) and fathers (15%) than reported in the general Dutch population (respectively, p < 0.001 and p < 0.05)., Conclusions: We report a clinically relevant impact of parenting an MPS III patient on psychosocial functioning, which is demonstrated by high levels of anxiety, depression, distress, and a remarkably high prevalence of PTSD. Structural monitoring of the psychosocial functioning of MPS III parents is therefore essential and may be beneficial for the whole family.

Background: Identification of children at risk for psychosocial problems is important to be able to provide supportive and tailored care at an early stage. Due to its brevity and wide age range, the Strengths and Difficulties Questionnaire (SDQ) Parent Form is an appropriate instrument for use in paediatric clinical practice as it facilitates assessment of psychosocial functioning from young childhood into adulthood. The aim of the present study was to provide Dutch normative data for the SDQ Parent Form., Methods: A sample of 1947 parents with children aged 2-18 years was drawn from a large panel of a Dutch research agency, stratified on Dutch key demographics of the parents. The SDQ Parent Form assesses the child's Emotional symptoms, Conduct problems, Hyperactivity-Inattention, Peer problems and Prosocial behaviour. Summary scores can be calculated: Internalising, Externalising and Total difficulties. Internal consistency (Cronbach's alpha coefficient) and normative scores (mean, median, clinical cut-off scores) of the SDQ- Parent Form were calculated in four age-groups 2-3, 4-5, 6-11 and 12-18 years. Gender differences were tested with independent t-tests., Results: A total of 1174 parents (60.3%) completed the SDQ. In the age-groups 2-3 and 4-5, norm scores are not available for Conduct problems and Peer problems due to insufficient internal consistency. In addition, in age-group 2-3, norm scores for Emotional symptoms and Internalising are not available because of insufficient internal consistency. In the age-groups 6-11 and 12-18, norm scores are available for all scales, with Cronbach's alpha coefficients 0.53-0.86. The comparison by gender revealed that boys had more behavioural problems than girls (0.000 < p < 0.048), most prevalent for Hyperactivity-Inattention, Peer Problems, Prosocial behaviour, Externalising and Total Difficulties., Conclusions: Dutch normative data by age-group and gender are now available for parent-reported SDQ scores in children aged 2-18 years. Due to insufficient internal consistency, normative scores for 2-5 year-old children could not be presented for several SDQ scales. Yet, the SDQ Total score provides a reliable indication of the psychosocial functioning of younger children. In case of high Total scores in children younger than 6 years, alternatively to scale scores, the answers on the individual items could yield useful clinical information about the child's problems.

Background: To provide targeted support to parents of children with DS, knowledge of their distress and everyday problems is crucial. For this purpose, psychosocial screening instruments can be a valuable addition to routine clinical practice., Aims: To determine differences on a psychosocial screener concerning distress and everyday problems in parents of young adolescents (YAs) with DS versus control parents and in mothers of YAs with DS versus fathers., Methods and Procedures: We compared outcomes of the Distress Thermometer for Parents in 76 mothers and 44 fathers of 11-13-year-olds with DS versus 64 mothers and 52 fathers of age-matched children without DS (comparing mothers and fathers separately). Additionally, we compared mothers and fathers within 34 parent couples of YAs with DS., Outcomes and Results: Clinical distress was not more frequent than in control parents. Mothers further did not report more everyday problems and only differed from their controls on one problem domain and some problem items. Fathers, however, reported more problems than their controls across most domains and wished to talk to a professional about their situation more frequently. Outcomes in mothers and fathers within parent couples did not differ significantly., Conclusions and Implications: This is one of few studies to report on the use of psychosocial screening instruments in parents of children with DS. Our results suggested that attention for fathers of YAs with DS is required. Psychosocial screening instruments that inquire about specific problems and the wish for referral can play an important role in achieving this., (Copyright © 2017 Elsevier Ltd. All rights reserved.)

Background: The purpose of this study is to provide Dutch normative data and to assess internal consistency and known-groups validity for the TNO AZL Preschool Children Quality of Life (TAPQOL) and the acute version of the generic Pediatric Quality of Life Inventory (PedsQL 4.0) in Dutch young children aged 0-7 years., Methods: Participants were selected from a panel of a large Dutch market research agency. A sample of 794 parents (response rate 61%, 39% fathers) of children (53% boys) from the general Dutch population, completed an electronic version of the TAPQOL (N = 227 infants aged 0-1 years) or PedsQL 4.0 (N = 293 toddlers aged 2-4 years and N = 274 young children aged 5-7 years)., Results: Except for the 'stomach' scale (α = .39), the TAPQOL showed acceptable to excellent internal consistency (α = .60-.88). The PedsQL 4.0 showed acceptable to excellent reliability in children aged 2-4 years (α = .60-.88) and in children aged 5-7 years (α = .76-.90). Children with a chronic health condition had lower scores than healthy children on 3 out of 12 domains of the TAPQOL (p = .001-.013) and on 2 out of 6 domains of the PedsQL 4.0 for children aged 2-4 years (p = .016-.04). The PedsQL 4.0 differentiated on all domains (p < .05) between children aged 5-7 years with and without a chronic health condition., Conclusion: In Dutch children aged 0-7 years old, HRQoL can be relialy measured with the TAPQOL and the PedsQL 4.0. However, it remains unclear whether these HRQoL instruments can distinguish between healthy children and children with a chronic health condition under the age of 5.

Background: Facilitating and enhancing interaction between stakeholders involved in the policymaking process to stimulate collaboration and use of evidence, is important to foster the development of effective Health Enhancing Physical Activity (HEPA) policies. Performing an analysis of real-world policymaking processes will help reveal the complexity of a network of stakeholders. Therefore, the main objectives were to unravel the stakeholder network in the policy process by conducting three systems analyses, and to increase insight into the similarities and differences in the policy processes of these European country cases., Methods: A systems analysis of the local HEPA policymaking process was performed in three European countries involved in the 'REsearch into POlicy to enhance Physical Activity' (REPOPA) project, resulting in three schematic models showing the main stakeholders and their relationships. The models were used to compare the systems, focusing on implications with respect to collaboration and use of evidence in local HEPA policymaking. Policy documents and relevant webpages were examined and main stakeholders were interviewed., Results: The systems analysis in each country identified the main stakeholders involved and their position and relations in the policymaking process. The Netherlands and Denmark were the most similar and both differed most from Romania, especially at the level of accountability of the local public authorities for local HEPA policymaking. The categories of driving forces underlying the relations between stakeholders were formal relations, informal interaction and knowledge exchange., Conclusions: A systems analysis providing detailed descriptions of positions and relations in the stakeholder network in local level HEPA policymaking is rather unique in this area. The analyses are useful when a need arises for increased interaction, collaboration and use of knowledge between stakeholders in the local HEPA network, as they provide an overview of the stakeholders involved and their mutual relations. This information can be an important starting point to enhance the uptake of evidence and build more effective public health policies.

This patient-reported outcome (PRO) study reports on 102 children with Robin sequence (RS) and their parents. There has been differentiated between those with isolated RS and those with RS as part of a syndrome, and take various treatments into account. All RS families from an earlier described cohort were invited to participate. Parents and RS children completed online questionnaires regarding health-related quality of life (HRQoL), satisfaction with appearance, parental distress, and RS specific topics. Results were compared with the Dutch norm population if available. There was no major difference in HRQoL in RS children and the Dutch norm population, nor between children with isolated RS and those with RS as part of a syndrome. The latter is likely due to the large percentage of children with Stickler syndrome, and small number of RS children with intellectual disability. Parental distress was higher in RS children with syndromes compared to parents of isolated RS children. When comparing various treatments, the subgroup treated by mandibular distraction showed a tendency of lower HRQoL scores, less satisfaction with appearance, and more parental distress. Also in the NPA group parents showed a tendency of more parental distress. Subgroups for each treatment were very small, however, and firm conclusions cannot be drawn. In this study, HRQoL in RS children is demonstrated comparable to the norm population, despite variations in treatment, possibly with less favorable outcome for children who received mandibular distraction. Markedly larger studies are needed to allow more reliable comparison of PROs in various treatments, and to incorporate PROs in management guidelines to obtain optimal patient care. © 2016 Wiley Periodicals, Inc., (© 2016 Wiley Periodicals, Inc.)

Purpose: The aim of this study was to provide Dutch normative data for the Distress Thermometer for Parents (DT-P) and to assess internal consistency and known-groups validity., Methods: A sample of 1421 parents (60.7 % mothers), representative of the Dutch population, completed online sociodemographic questionnaire and the DT-P, which includes a thermometer (0 (no distress) to 10 (extreme distress), ≥4 clinically elevated distress) and everyday problems across six problem domains (practical, social, emotional, physical, cognitive, and parenting). Internal consistency was calculated using Cronbach's alphas. Known-groups validity was assessed by comparing parents of a child with a chronic condition (N = 287, 20.2 %) with parents of healthy children, using Mann-Whitney U tests and Chi-square tests., Results: The DT-P showed acceptable internal consistency (Cronbach's alphas = .52-.89). Parents of a child with a chronic condition more often reported clinically elevated distress than parents of healthy children (53.0 versus 38.2 %, p < .001). Also, on all domains they reported more problems (p = .000-.022). Normative scores for mothers and fathers separately were provided., Conclusion: The DT-P distinguishes well between parents of a child with and without a chronic condition. With the current norms available, distress can be evaluated in parents of a child with a chronic condition compared to parents of healthy children in pediatric clinical practice., Competing Interests: All authors declare that they have no conflict of interest. Ethical approval All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards. This article does not contain any studies with animals performed by any of the authors. Informed consent Informed consent was obtained from all individual participants included in the study.

Decolonization has brought political independence to half the Caribbean states in the last half of the 20th century, while the other states remain affiliated. Previous studies suggested a beneficial impact of affiliated status on population health, which may be mediated by more favorable economic development. We assessed how disparities in life expectancy between currently sovereign and affiliated states developed over time, whether decolonization coincided with changes in life expectancy, and whether decolonization coincided with similar changes in GDP per capita. Time-series data on life expectancy and related variables, GDP per capita and political status were collected from harmonized databases. We quantified variations in life expectancy by current political status during the 1950-2010 period. We assessed whether decolonization coincided with life expectancy trend changes by: 1. calculating the annual changes before and after independence, and 2. evaluating trend breaks in a predefined period during decolonization using joinpoint analyses. Similar analyses were undertaken for GDP per capita. Life expectancy in currently sovereign Caribbean states was already lower than in affiliated states before political independence. Overall, decolonization coincided with reductions in life expectancy growth, but not with reductions in economic growth, and changes in life expectancy growth in the decade after independence did not correspond with changes in economic performance. The widening of the life expectancy gap between currently sovereign and affiliated states accelerated in the 1990's and continues to increase. Despite considerable life expectancy gains in all Caribbean states, life expectancy in currently sovereign states increasingly lags behind that of states which remained affiliated. Our results indicate that changing economic conditions were not the main determinant of the unfavorable trends in life expectancy during and after decolonization. Circumstantial evidence points to the weakening of bureaucracies during decolonization underlying the uneven life expectancy developments of currently affiliated and sovereign states., (Copyright © 2016 Elsevier Ltd. All rights reserved.)

Background: The Dutch Public Health Status and Forecasts report (PHSF Report) integrates research data and identifies future trends affecting public health in the Netherlands. To investigate how PHSF contributions to health policy can be enhanced, we analysed the development process whereby the PHSF Report for 2010 was produced (PHSF-2010)., Method: To collect data, a case study approach was used along the lines of Contribution Mapping including analysis of documents from the PHSF-2010 process and interviews with actors involved. All interviews were recorded and transcribed ad verbatim and coded using an inductive code list., Results: The PHSF-2010 process included activities aimed at alignment between researchers and policy-makers, such as informal meetings. However, we identified three issues that are easily overlooked in knowledge development, but provide suggestions for enhancing contributions: awareness of divergent; continuously changing actor scenarios; vertical alignment within organizations involved and careful timing of draft products to create early adopters., Conclusion: To enhance the contributions made by an established public health report, such as the PHSF Report, it is insufficient to raise the awareness of potential users. The knowledge product must be geared to policy-makers' needs and must be introduced into the scenarios of actors who may be less familiar. The demand for knowledge product adaptations has to be considered. This requires continuous alignment efforts in all directions: horizontal and vertical, external and internal. The findings of this study may be useful to researchers who aim to enhance the contributions of their knowledge products to health policy., (© The Author 2016. Published by Oxford University Press on behalf of the European Public Health Association.)

Background: To improve knowledge utilization in policymaking, alignment between researchers and policymakers during knowledge production is essential, but difficult to maintain. In three previously reported case studies, we extensively evaluated complex research projects commissioned by policymakers to investigate how alignment is achieved in a research process and to discover ways to enhance knowledge contributions to health policy. In the present study, we investigated how the findings of these three research projects could be integrated into a practical tool for researchers to enhance their contribution to evidence-based policy., Methods: A cross-case analysis was conducted to integrate the findings of the evaluation of the three research projects and to identify important alignment areas in these projects. By means of an iterative process, we prepared a tool that includes reflection questions for researchers. The "Research for Policy" tool was tested with input from the project managers of three new research projects. Based on the findings, the final version of the Research for Policy tool was prepared., Results: By cross-case analysis of the three case studies, the following important alignment areas were identified: the goal, quality, relevance, timing, and presentation of research, the tasks and authorities of actors, the consultative structure and vertical alignment within organizations, and the organizational environment. The project managers regarded the Research for Policy tool as a useful checklist for addressing the important alignment areas in a research project. Based on their feedback, the illustrative examples from the case studies were added to the reflection questions. The project managers suggested making the tool accessible not only to researchers but also to policymakers. The format of the Research for Policy tool was further adjusted to users' needs by adding clickable links., Conclusions: Alignment between research and policymaking requires continuous efforts and a clear understanding of process issues in the research project. The Research for Policy tool offers practical alignment guidance and facilitates reflection on process issues, which supports researchers in aligning with policymakers and in acting in a context-sensitive way.

Background: Despite the acknowledged value of an EU health information system (EU-HISys) and the many achievements in this field, the landscape is still heavily fragmented and incomplete. Through a systematic analysis of the opinions and valuations of public health stakeholders, this study aims to conceptualize key features of an EU-HISys., Methods: Public health professionals and policymakers were invited to participate in a concept mapping procedure. First, participants (N = 34) formulated statements that reflected their vision of an EU-HISys. Second, participants (N = 28) rated the relative importance of each statement and grouped conceptually similar ones. Principal Component and cluster analyses were used to condense these results to EU-HISys key features in a concept map. The number of key features and the labelling of the concept map were determined by expert consensus., Results: The concept map contains 10 key features that summarize 93 statements. The map consists of a horizontal axis that represents the relevance of an 'organizational strategy', which deals with the 'efforts' to design and develop an EU-HISys and the 'achievements' gained by a functioning EU-HISys. The vertical axis represents the 'professional orientation' of the EU-HISys, ranging from the 'scientific' through to the 'policy' perspective. The top ranking statement expressed the need to establish a system that is permanent and sustainable. The top ranking key feature focuses on data and information quality., Conclusions: This study provides insights into key features of an EU-HISys. The results can be used to guide future planning and to support the development of a health information system for Europe., (© The Author 2015. Published by Oxford University Press on behalf of the European Public Health Association. All rights reserved.)

Objectives: The Dutch Health Care Performance Report, issued by the National Institute of Public Health and the Environment, aims to monitor health care performance in The Netherlands. Both the National Institute and the Ministry of Health wish to increase the contribution of the Report to health care policy-making. Our aim was to identify ways to achieve that., Method: We used contribution mapping as a theoretical framework that recognizes alignment of research as crucial to managing contributions to policy-making. To investigate which areas need alignment efforts by researchers and/or policy-makers, we interviewed National Institute researchers and policy-makers from the Ministry of Health and assessed the process for developing the 2010 Report., Results: We identified six areas where alignment is specifically relevant for enhancing the contributions of future versions of the Dutch Health Care Performance Report: well-balanced information for different ministerial directorates; backstage work; double role actors; reports of other knowledge institutes; data collection/generation and presentation forms., Conclusion: The contribution of health care performance reporting to policy-making is complex and requires continuous alignment efforts between researchers and policy-makers. These efforts should form an inseparable part of health care performance reporting and although this demands considerable resources, it is worth considering since it may pay back in better contributions to policy-making., (© The Author(s) 2015.)

Background: Contemporary research should increasingly be carried out in the context of application. Nowotny called this new form of knowledge production Mode-2. In line with Mode-2 knowledge production, the Dutch government in 2006 initiated the so-called Academic Collaborative Centres (ACC) for Public Health. The aim of these ACCs is to build a regional, sustainable knowledge-sharing network to deliver socially robust knowledge. The present study aims to highlight the enabling and constraining push and pull factors of these ACCs in order to assess whether the ACCs are able to build and strengthen a sustainable integrated organizational network between public health policy, practice, and research., Methods: Our empirical analysis builds on a mixed methods design. Quantitative data was derived from records of a survey sent to all 11 ACCs about personnel investments, number and nature of projects, and earning power. Qualitative data was derived from 21 in-depth interviews with stakeholders involved. The interviews were tape-recorded, transcribed, and manually coded as favourable or unfavourable pull or push factors., Results: The extra funding appeared to be the most enabling push factor. The networks secured external grants for about 150 short- and long-term Mode-2 knowledge production projects in the past years. Enabling pull factors improved, especially the number of policy-driven short-term research projects. Exchange agents were able to constructively deal with the constraining push factors, like university's publication pressure and budget limitations. However, the constraining pull factors like local government's involvement and their low demand for scientific evidence were difficult to overcome., Conclusions: A clear improvement of the organizational networks was noticed whereby the ACC's were pushed rather than pulled. Efforts are needed to increase the demand for scientific and socially robust evidence from policymakers and to resolve the regime differences between the research and policy systems, in order to make the bidirectionality of the links sustainable.

Purpose: The aim of this study was to assess internal consistency and construct validity (known-groups validity) and to provide Dutch norm data for the Dutch Pediatric Quality of Life Inventory Multidimensional Fatigue Scale for Young Adults ages 18-30 years (PedsQL fatigue&#95;YA)., Methods: A Dutch sample of 649 young adults completed online a sociodemographic questionnaire and the PedsQL fatigue&#95;YA including three subscales: general fatigue, sleep/rest fatigue and cognitive fatigue (0-100: Higher scores indicate less fatigue symptoms)., Results: The PedsQL fatigue&#95;YA showed satisfactory to good internal consistency (Cronbach's alpha = .70-.94), except for one scale (.68). The mean scale scores were 68.23 (SD 19.15) for 'general fatigue,' 67.04 (SD 15.54) for 'sleep/rest fatigue' and 74.62 (SD 19.02) for 'cognitive fatigue.' Men reported significantly higher scores on 'general fatigue' and 'sleep/rest fatigue' than women. The PedsQL fatigue&#95;YA distinguished between healthy young adults and young adults with chronic health conditions, with higher scores on all scales in healthy young adults than in those with a chronic health condition., Conclusion: The results demonstrate good psychometric properties of the PedsQL fatigue&#95;YA in a sample of Dutch young adults. With the current norms available, it is possible to evaluate fatigue in the Netherlands from childhood to adulthood with the PedsQL Multidimensional Fatigue Scale.

We aimed to determine the levels of anxiety and depression in mothers and fathers of a chronically ill child (0-18 years) and to study which parental and child variables are associated with anxiety and depression. In a cross-sectional design, anxiety and depression were assessed with the Hospital Anxiety and Depression Scale. Scores were compared to a Dutch reference group by analysis of variance and logistic regression analysis. Linear regression analyses were performed to examine which variables were associated with anxiety and depression. Mothers of a chronically ill child (n = 566) scored significantly higher than the reference group (p < .001) on anxiety (Mean 5.9 vs 4.8) and depression (Mean 4.5 vs 3.1). Fathers (n = 123) had higher depression scores (Mean 4.5 vs 3.6; p < .05), but fathers' anxiety scores were comparable to the reference group. The percentages of mothers in the clinical range of anxiety (31.8 vs 20.7 %, OR 2.03, 95 % CI 1.46-2.83) and depression (23.0 vs 12.0 %, OR 2.76, 95 % CI 1.84-4.13) were higher (p < .001) than in the reference group. No differences were found for fathers in the clinical range for anxiety and depression. Practical problems in daily life (a: β = .33, d: β = .25) and parenting stress (a: β = .30, d: β = .32) showed the strongest association with anxiety and depression for parents as a group. Illness-related characteristics of the child were not related. Parents of a chronically ill child, especially mothers, reported high levels of anxiety and depression. Awareness about parental anxiety and depression in pediatrics is important as well as targeted interventions.

Background: There is much evidence that parents have an influence on the alcohol use of their children. However, in general the relationship is rather weak. A reason for this small association may be due to the fact that adolescents are a heterogeneous group and that, consequently, the association between the quality of the parent-child relationship and alcohol use varies for diverse subgroups, resulting in an overall small effect. In an earlier study we found five different segments for adolescents regarding their attitude towards alcohol. This article reports on a study into the differences between these segments with respect to the quality of the parent-child relationship and parental attitudes to alcohol. Moreover, we examined segment-specific associations of the quality of the parent-child relationship and alcohol use., Methods: This study used data from a survey held among adolescents aged 12 to 18. A random sample of 59,073 adolescents was drawn from 67 municipalities in the south of the Netherlands. To assign respondents into one of the five segments, a questionnaire of 28 items concerning alcohol and approval from others from the original segmenting study was included in the internet version. Therefore, only the results of the internet version (N = 12,375 adolescents) were analysed., Results: Both the quality of the parent-child relationship and the attitude of the parents towards the drinking behaviour of their children differed between the segments. Significant associations were found between the quality of the parent-child relationship and life-time and recent alcohol use and binge drinking. The interaction between the quality of the parent-child relationship and the segments was only significant for binge drinking., Conclusions: The quality of the parent-child relationship seemed to be most strongly associated with life-time alcohol use, suggesting that parents appear to play the most important role in the prevention of alcohol use. Moreover, the results showed segment-specific associations between the quality of the parent-child relationship and binge drinking, indicating that the role of parents in heavy drinking is different for the various segments.

Background: A chronic illness, such as Juvenile Idiopathic Arthritis (JIA), has an impact on the whole family, especially on parents caring for the ill child. Therefore the aim of this study is to evaluate parental Health Related Quality of Life (HRQOL) and parental perceptions of child vulnerability (PPCV) and associated variables in parents of a child with JIA., Methods: Parents of all JIA patients (0-18 years) in Amsterdam, the Netherlands, were eligible. HRQOL was measured using the TNO-AZL Questionnaire (TAAQOL) and PPCV using the Child Vulnerability Scale (CVS). The HRQOL of parents of a child with JIA was compared to a norm population, and differences between parents of a child with JIA and active arthritis versus parents of a child with JIA without active arthritis were analyzed (ANOVA). For PPCV, parents of a child with JIA were compared to a norm population, including healthy and chronically ill children (Chi(2), Mann-Whitney U test). Variables associated with PPCV were identified by logistic regression analyses., Results: 155 parents (87.5% mothers) completed online questionnaires. JIA parents showed worse HRQOL than parents of healthy children on one out of twelve domains: fine motor HRQOL (p < .001). Parents of children with active arthritis showed worse HRQOL regarding daily activities (p < .05), cognitive functioning (p < .01) and depressive emotions (p < .05) compared to parents of children without active arthritis. Parents of children with JIA perceived their child as more vulnerable than parents of a healthy child (p < .001) and parents of a chronically ill child (p < .001). Parents of children with active arthritis reported higher levels of PPCV (p < .05) than parents of children without active arthritis. A higher degree of functional disability (p < .01) and shorter disease duration (p < .05) were associated with higher levels of PPCV., Conclusion: The HRQOL of JIA parents was comparable to the HRQOL of parents of a healthy child. JIA parents of a child with active arthritis showed worse HRQOL than parents of a child without active arthritis. Parents perceived their child with JIA as vulnerable.

Aim: Kawasaki disease (KD) is an acute paediatric vasculitis. The psychosocial consequences of this sudden illness for parents are unknown. This study aimed to evaluate health related quality of life (HRQOL) and parental perceptions of child vulnerability (PPCV) in parents of children with KD, and to identify variables associated with PPCV., Methods: This cross-sectional study included 288 parents (83% mothers) of KD patients (mean age 8.7 years). HRQOL was assessed using the TNO-AZL Questionnaire for Adult's HRQOL (TAAQOL) and PPCV using the Child Vulnerability Scale (CVS). Scores of KD parents were compared with reference groups of Dutch parents. Logistic regression analyses were performed to examine associated variables., Results: The HRQOL of KD parents was comparable to the HRQOL of parents of healthy children. However, KD parents showed significantly higher PPCV, regarding both the median CVS total score and the percentage in the clinical range. No differences were found in CVS outcomes between KD parents and parents of a chronically ill child. None of the studied parental, child and disease characteristics were significantly associated with PPCV., Conclusion: Parents perceived their KD child more vulnerable to illness than healthy children, while in reality the majority had fully recovered from KD., (©2014 Foundation Acta Paediatrica. Published by John Wiley & Sons Ltd.)